Xanthogranulomatous osteomyelitis of the jaw in a young boy: a case report

Xanthogranulomatous inflammation is a rare chronic inflammatory condition. It has been described in the long bones, and can affect various organs, including the salivary glands, gallbladder, kidneys, and gastrointestinal tract. The diagnosis is made throught histopathology which reveales foamy macrophages alongside polymorphonuclear leukocytes, plasma cells, and polyclonal lymphocytes arranged in a mosaic-like pattern. Here, we present the case of a 16-year-old boy in good general health who was referred by his orthodontist to the oral surgery department at Bretonneau Hospital for an asymptomatic multipartite image of the left mandibular angle that was discovered incidentally on a panoramic radiograph. The clinical examination was unremarkable. Cone beam computed tomography revealed a multipartite osteolytic lesion of the left mandibular angle, but this that was not specific enough for a diagnosis. Magnetic resonance imaging ruled out a vascular malformation. A biopsy revealed xanthogranulomatous osteomyelitis. In summary, this is the second case of xanthogranulomatous osteomyelitis localised to the mandible. This highlights a crucial point : radiological images of xanthogranulomatous osteomyelitis do not allow for a diagnosis; a biopsy is essential to rule out an aggressive or a malignant tumor.

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Publication Details

Journal
Orphanet Journal of Rare Diseases
Published
2025-09-24
DOI
https://doi.org/10.1186/s13023-025-04010-w
Citations
1
Primary Topic
Infectious Disease Case Reports and Treatments
Type
article
Field-Weighted Citation Impact
1.40
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article

Xanthogranulomatous osteomyelitis of the jaw in a young boy: a case report

A.-L. Ejeil, Dridi Sophie-Myriam, Bloch-Maier Kevin, Sulukdjian Arek
1 citations
Orphanet Journal of Rare Diseases
Infectious Disease Case Reports and Treatments
1.40
article

Xanthogranulomatous osteomyelitis of the jaw in a young boy: a case report

A.-L. Ejeil, Dridi Sophie-Myriam, Bloch-Maier Kevin, Sulukdjian Arek
article en
1 citations

Abstract

Xanthogranulomatous inflammation is a rare chronic inflammatory condition. It has been described in the long bones, and can affect various organs, including the salivary glands, gallbladder, kidneys, and gastrointestinal tract. The diagnosis is made throught histopathology which reveales foamy macrophages alongside polymorphonuclear leukocytes, plasma cells, and polyclonal lymphocytes arranged in a mosaic-like pattern. Here, we present the case of a 16-year-old boy in good general health who was referred by his orthodontist to the oral surgery department at Bretonneau Hospital for an asymptomatic multipartite image of the left mandibular angle that was discovered incidentally on a panoramic radiograph. The clinical examination was unremarkable. Cone beam computed tomography revealed a multipartite osteolytic lesion of the left mandibular angle, but this that was not specific enough for a diagnosis. Magnetic resonance imaging ruled out a vascular malformation. A biopsy revealed xanthogranulomatous osteomyelitis. In summary, this is the second case of xanthogranulomatous osteomyelitis localised to the mandible. This highlights a crucial point : radiological images of xanthogranulomatous osteomyelitis do not allow for a diagnosis; a biopsy is essential to rule out an aggressive or a malignant tumor.

Orphanet Journal of Rare DiseasesVol. 20(1)
Hôpital Beaujon (FR), Centre Hospitalier Universitaire de Nice (FR), Assistance Publique – Hôpitaux de Paris (FR), Hôpital Bretonneau (FR)
Openalex Percentile: Top 16%
Infectious Disease Case Reports and Treatments
1.40
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Xanthogranulomatous osteomyelitis of the jaw in a young boy: a case report — A.-L. Ejeil, Dridi Sophie-Myriam, et al. · Orphanet Journal of Rare Diseases (2025) | TGRS Research Map | TGRS